Articles
HD-PRO-TRIAD™ Validation: A Patient-reported Instrument for the Symptom Triad of Huntington’s Disease
Authors:
- Noelle E. CarlozziEmail Noelle E. Carlozzi
- David Victorson
- Victor Sung
- Jennifer L. Beaumont
- Wendy Cheng
- Brian Gorin
- Mei Sheng Duh
- David Samuelson
- David Tulsky
- Sandra Gutierrez
- Cindy J. Nowinski
- Allison Mueller
- Vivienne Shen
- Samuel Frank
Abstract
Background: Few valid, disease-specific measures of health-related quality of life (HRQOL) capture the spectrum of symptoms associated with Huntington’s disease (HD). The HD-PRO-TRIAD™ is a new, HD-specific, patient-reported outcome (PRO) instrument of the HD symptom triad (cognitive decline, emotional/behavioral dyscontrol, and motor dysfunction) designed for clinical research and practice. The objective was to validate the HD-PRO-TRIAD™ through a cross-sectional sample of individuals with HD and caregivers.
Methods: Development of the HD-PRO-TRIAD™ has been described elsewhere. A total of 132 individuals with HD and 40 HD caregivers, comprising 29 dyads, participated in the cross-sectional psychometric validation of this instrument. Participants provided responses to the HD-PRO-TRIAD™ and other HRQOL and disease severity instruments (EuroQOL 5D, Short Form 12, Neuro-QOL Item Banks, PROMIS Global Health, and self-reported Unified Huntington’s Disease Rating Scale Total Functional Capacity and Independence Scales). Internal consistency, construct validity, and patient–caregiver proxy consistency were evaluated.
Results: Internal consistency of the three domains and overall HD-PRO-TRIAD™ instrument was supported by Cronbach’s alpha values $0.94. Construct validity was supported by significant correlations between HD-PRO-TRIAD™ domain scores and other measures of the same domains (e.g., significant positive correlations between HD-PRO-TRIAD™ Anxiety with Neuro-QOL Anxiety), as well as slightly weaker but still strong correlations with other HRQOL instruments (e.g., HD-PRO-TRIAD™ Anxiety and UHDRS Independence; all p<0.01). Consistency between patient self-report and caregiver proxy report was supported by an intra-class correlation coefficient ≥0.92 for all three domains and the overall instrument.
Discussion: These data indicate that HD-PRO-TRIAD™ is a reliable and valid HRQOL instrument that captures the typical triad of HD symptoms.
- Year: 2014
- Volume: 4
- Page/Article: 223
- DOI: 10.5334/tohm.177
- Submitted on 23 Jan 2014
- Accepted on 26 Feb 2014
- Published on 14 Apr 2014
- Peer Reviewed